Anomalous Origin of Left Coronary Artery with Aortopulmonary Window - A Case Report with Surgical Correction and Delayed Control

Abstract

A 9-month-old male baby was investigated for a massive left-to-right shunt at the arterial level. On right heart catheterization a patent ductus could not be demonstrated but retrograde arterial angiography showed an aorto-pulmonary window. During corrective surgery on cardiac bypass, the left coronary artery was found to arise from the aorto-pulmonary window, this was corrected by dividing the communication distal to the origin of the left coronary artery. On follow-up investigation 6 months after operation the baby was without symptoms and showed a fully functional left coronary artery. To our knowledge this is the first case of such a congenital anomaly to be described.

Pre-operative identification of an anomalous left coronary artery from the pulmonary artery in association with tetralogy of Fallot and an aortopulmonary window

“…2 Interestingly, however, all four previous cases of complex aortopulmonary window in association with an anomalous coronary arterial origin were only diagnosed at the time of surgical intervention, with this patient being the first to have had this diagnosis before surgery. [1][2][3][4] The precise mechanism of the haemodynamic compromise and myocardial ischaemia in this case is not clear. In the presence of an aortopulmonary window, one would assume that the anomalous left coronary perfusion would be maintained.…”

Unique Association of Aortopulmonary Window With Anomalous Origin of Left Coronary Artery From Pulmonary Artery in an Infant: A Blessing in Disguise?

“…Since the first report by Bourlon et al only 12 patients have been reported in literature with this unusual combination of APW and ALCAPA. 2–4 Of these, preoperative identification of ALCAPA was accomplished in one patient only, suggesting the challenges in echocardiographic identification of this coronary anomaly in the presence of associated PAH, the hemodynamic reasons for which would be discussed in further detail below. Failure to identify and correct this coronary anomaly during surgery for associated lesions would result in unforeseen postoperative ventricular dysfunction.…”

“…Since the first report by Bourlon et al only 12 patients have been reported in literature with this unusual combination of APW and ALCAPA. [2][3][4] Of these, preoperative identification of ALCAPA was accomplished in one patient only, suggesting the challenges in Submitted August 20, 2021; Accepted October 28, 2021.…”

Prenatal Diagnosis of an Aorto-Pulmonary Window by Fetal Echocardiography

“…Kutsche and Van Mierop [3] reported 52% of cardiovascular malformations in a series of 188 APWs. These were mainly VSD and ASD [3,7], interruption of the aortic arch [1,3], patent ductus arteriosus [3], aortic valve atresia [8], coarctation of the Ao [1,3], aortic origin of the right PA [3,9], tetralogy of Fallot [3,10], anomalous origin of the coronary arteries [3,8], aortic or pulmonary valve stenosis [3], and Berry's syndrome associating distal APW, interrupted aortic arch, and aortic origin of the right PA [9,11]. The prognosis of APW depends on these associated cardiac malformations.…”